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BrazJOtorhinolaryngol.2016;82(1):112---115

www.bjorl.org

Brazilian

Journal

of

OTORHINOLARYNGOLOGY

CASE

REPORT

Giant

sialolith

of

submandibular

gland

duct

treated

by

excision

and

ductal

repair:

a

case

report

,

夽夽

Sialolito

gigante

de

ducto

da

glândula

submandibular

tratado

por

excisão

e

reparo

ductal:

relato

de

caso

Thiago

de

Paula

Oliveira

a

,

Isaac

Nilton

Fernandes

Oliveira

a

,

Eduardo

Carvalho

Paes

Pinheiro

a

,

Renata

Caroline

Ferreira

Gomes

a

,

Pietro

Mainenti

b,c,∗

aFaculdadedeMedicinadeJuizdeFora,UniversidadePresidenteAntônioCarlos,JuizdeFora,MG,Brazil

bDepartmentofPathology,FaculdadedeMedicinadeJuizdeFora,UniversidadePresidenteAntônioCarlos,JuizdeFora,MG,

Brazil

cDepartmentofOralandMaxillofacialSurgery,CentroMédicoRioBranco,JuizdeFora,MG,Brazil

Received11March2015;accepted27March2015 Availableonline7September2015

Introduction

Sialolithiasis is one of the most common diseases of the salivary glands.1,2 It is a condition characterized by an

obstructivephenomenoninasalivaryglandorinits

excre-tory duct due to a calculus.1 The clinical presentation is

usuallycharacterized by local swelling,pain,infection of

theaffectedarea,anddilationofthesalivaryduct.1

Sialo-lithiasisusuallyaffectsadultsbetweenthethirdandfourth

decades of life, with a frequency of 12:1000.3 The

num-berofcasesinmalepatientsisabouttwicethatoffemale

patients.3Itisestimatedthat80---90%ofcasesoccurinthe

Pleasecitethisarticleas:OliveiraTP,OliveiraINF,PinheiroECP, GomesRCF,MainentiP.Giantsialolithofsubmandibularglandduct treatedbyexcisionandductalrepair:casereport.BrazJ Otorhino-laryngol.2016;82:112---5.

夽夽Institution:FaculdadedeMedicinadeJuizdeFora, Universi-dadePresidenteAntônioCarlos,JuizdeFora,MG,Brazil.

Correspondingauthor.

E-mail:pietromainenti@terra.com.br(P.Mainenti).

submandibular gland, while 10---20% occur in the parotid

gland.3Thesizeofthecalculi variesfrom<1mmtoafew

centimeters.Althoughthefrequencyofsialolithiasisis

rel-ativelyhigh,theoccurrenceofgiantsialoliths, largerthan

1.5cminanydiameter,israre.Forthisreasonfewstudies

arefoundinthepertinentmedicalliterature.1,4

This report describes a case of giant sialolith in a

42-years-oldmale,addressingtheclinicalfeatures,the

diag-nosis, and theductal repair surgeryperformed to restore

salivaryflow.

Case

report

The patient,a 42-year-old black man,attended a dental

appointment in Marchof 2014. Afterroutine radiographic

examination,hewasreferredforaconsultationwithanoral

andmaxillofacialsurgeon,inAprilof2014.During

anamne-sisthe patientdenied anyprevious diseases.He reported

only an uneventfulsurgery on the right leg. The physical

examinationshowedanankyloglossiaand,duringpalpation,

ahardnessintherightsubmandibularsalivarygland.To

fur-ther investigate the case, imaging exams wererequested

(Fig.1A).Aprovisionaldiagnosisofsialolithiasisintheright

submandibularglandductwassuggested.

http://dx.doi.org/10.1016/j.bjorl.2015.03.013

(2)

Giantsialolithofsubmandibulargland:reportofexcisionandductalrepair 113

Figure1 (A)Computedtomographyscan(axialaspect)revealingamineralizedtissuewithheterogeneousdensityanddimensions of3.0×1.0cm,approximately.(B)Three-dimensionalimageofthesialolithandthemandible.

Sincethesialolithhadexuberantdimensions,anexcision

followedbythereconstructionofthesubmandibulargland

ductwasproposed.Bloodtestsandsurgicalriskexamswere

requestedforthepatient.

OnMay21,2014,thesurgicalprocedurewasconducted

by an intra-oral approach. The sialolith was removed by

curettage afterdirect incisionof theduct.A partial

min-eralizationfavoredthefragmentationofthedistalportion

ofthecalculus. Atruesalivaryglandcystwasremovedin

associationwiththecalculus(Fig.2).

Forthetreatmentofankyloglossia,atonguefrenectomy

wasperformed. To restore the salivaryflow, a No.8

ure-thral catheter wasplaced in the residual duct path. The

mucosawassuturedaroundthe catheterusingaVicryl

3-0suture inordertorepair theductof thesubmandibular

gland.

Theothertissuesweresuturedinanatomicalplanesand

therewerenocomplicationsduringthesurgicalprocedure.

Twodaysafterthesurgery,anultrasoundshowedthatthe

catheterwasinsidethesubmandibularglandduct(Fig.3).

Aftermilkingof thegland,thepresenceof crystalline

liq-uidflowingfromwithinthe tubewasnoted(Fig.4).Eight

daysafterthesurgery,thepatientreportedanincreasein

salivaryvolumeandtheoccurrence ofcontractionsin the

submandibularglandregion.

Figure3 Ultrasoundshowingthecatheterinsidethesalivary glandduct.

Thesutures andthedrainwereremovedfourteen days

afterthesurgery.Aglandmilkingmaneuvershowedcopious

salivation, indicating that the performed surgical

tech-niquesucceededinreconstructingtheductalstructure.The

(3)

114 OliveiraTPetal.

Table1 Comparativetableofconsultedcases.

Author Sialolithsize Symptoms Removalmethod Age Gender

Guptaetal. (Case1)

2.8cm×1.1cm Intermittent,dullachingpain,

andswellinginleft submandibularareaduring meals

Surgicallyremovedvia intraoralapproachunder localanesthesiaand transpositionofductal opening

48 Male

Guptaetal. (Case2)

1.9cm×5.0cm Swellinginmouthassociated

withpainoverleftsideofface duringintakeoffood

Surgicallyremovedvia intraoralapproachunder localanesthesiaand transpositionofductal opening

45 Female

Iqbaletal. 3.5cm×3.0cm Asymptomatic Surgeryunderlocal

anesthesia,intra-oral approachwith marsupialization

55 Male

Dalaletal. 1.8cm×6.0cm Pusdischargeandcontinuous

painofprickingandsharp nature,radiatingtothetongue withrestrictedtongue

movement

Sialolithotomyvia intraoralapproachunder localanesthesia

40 Female

Fowell& MacBean

4.1cm Painintherightfloorofmouth andsubmandibularregion, exacerbatedbyswallowing

Excisionoftheright submandibularglandand stoneviaastandard extra-oralapproach

58 Male

Krishnanetal. (Case1)

3.4cm Recurrentpainandswelling overeightyearsthatincreased duringmeals.Inthelasttwo yearspresentedasymptomatic

Sialolithotomyvia intraoralapproachunder localanesthesia.The woundwaslefttoheal bysecondaryintention

41 Male

Krishnanetal. (Case2)

2.5cm Multipleepisodesofpainand swellingintheleftlowerpart ofthemandible,duringthe pastfourtofiveyears, especiallyatmealtimes

Surgicallyremoved throughatransoral approach,withsharp dissectionunderlocal anesthesia

32 Female

follow-upappointments withintwo monthsof thesurgery showednocomplicationsorcomplaints.

Discussion

Sialolithiasis is a disease that can affect any age group, withahigherprevalenceinmaleadults.2,5Itmainlyaffects

Figure 4 The catheter and the sutures are in the correct surgicalplacement.

the submandibular gland.6 Despite being a common

dis-ease, the presence of giant calculus is extremely rare

and most sialoliths do not exceed 1.5cm.3,5 The

calcu-lus in the present case had dimensions of approximately

3.0cm×1.0cm,thusconsideredagiantsialolith.1

The reported symptoms usually are pain and swelling

in the gland, which worsen during the meal time

(Table1).2---4,6,7Inthecurrentreport,thepatientremained

asymptomatic despite the exuberant dimensions of the

stone.

AccordingtoJensen8andCawsonetal.,7salivarystones

canbeassociatedwiththepresenceof truesalivarygland

cysts.Suchlesionsoccurduetotheobstructionofthe

sali-varyflow,followedbyaproliferationoftheductepithelium

thatsurroundsthestone.Thepresentspecimenpresented

asquamousandoncocyticdifferentiationinaccordwiththe

literature.8

Thepathophysiologyofthestoneformationisstillpoorly

understood.3 However, it is believed that the sialolith is

formed after the deposition of calcium salts around a

‘‘niche’’oforganicmaterial.7

In 80% of cases the submandibular gland is affected7

(4)

Giantsialolithofsubmandibulargland:reportofexcisionandductalrepair 115

compositionofthe salivaproducedby thegland,whichis

morealkalineandwithamajorconcentrationofcalcium6;

(b) the salivaryflow occursagainst gravity2,9; and(c) the

longandtortuousanatomyoftheductofthesubmandibular

gland.6,9Allthesefactorsworktogetherintheformationof

thecalculusinthesubmandibulargland.2,6,9Intheauthors’

opinion, theoccurrence of the sialoliths presented in the

consultedliteratureisinlinewiththeirunderstanding.

Regarding the treatment, a less invasive procedure is

of utmost importance in order to preserve the gland’s

function.2,4,7,9 The pertinent literature indicated some

surgical procedures such as trans-oral sialolithotomy,

sialoendoscopy, extracorporeal shockwave lithotripsy, and

resectionofthegland.2,3Forsmallsialoliths, conservative

treatmentsusingsialogoguesandmassageoftheglandare

alsopossible.7Thecurrentcaseshowedthetreatmentofan

exuberant calculus throughan intra-oral approach

associ-atedwithaductalrepair.AlthoughFowelletal.2concluded

that sialoplasty is one of the main treatments for giant

sialoliths,thistechniquehasnotbeendescribedorusedby

theauthorsconsulted.Theyperformedtheremovalofthe

sialolithwithclosurebysecondaryintention.

Among the possible surgical complications, one is

injury of the mandibular nerve,2 another is Wharton’s

duct stenosis.2 There was no evidence of any of these

complicationsinthepresentcase.Theductalrepair

main-tainedsalivaryflowbetweentheglandandtheoralcavity.

Thesurgicalremovalofsialolithvariesbetweensurgeons.

Thepreferredapproachismostlyperformedthrough

intra-oralintervention(Table1).

Conclusion

The present case reportdescribed the removalof agiant

sialolith.To thebestoftheauthors’ knowledge,thiscase

isuniquewithregardtothesurgicalductalrepairafterthe

excisionofasalivarystone.

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

References

1.GuptaA,RattanD,Gupta R.Giantsialolithsofsubmandibular glandduct: reportoftwo caseswithunusualshape.Contemp ClinDent.2013;4:78---80.

2.FowellC,MacbeanA.Giantsalivarycalculiofthesubmandibular gland.JSurgCaseRep.2012;9:1---4.

3.IqbalA,GuptaAK,NatuSS,GuptaAK.Unusuallylargesialolith ofWharton’sduct.AnnMaxillofacSurg.2012;2:70---3.

4.DalalS,JainS,AgarwalS,VyasN.Surgicalmanagementofan unusuallylargesialolithofWharton’sduct:acasereport.King SaudUnivJDentSci.2013;4:33---5.

5.Filho MAO, Almeida LE, Pereira JA. Sialolito gigante asso-ciado à fístula cutânea. Rev Cir Traumatol Buco-Maxilo-Fac. 2008;8:35---8.

6.Branco BLC, Cardoso AB, Caubi AF, Pena GN. Sialolitíase: relatodeumcaso.RevCirTraumatolBuco-Maxilo-Fac.2003;3: 9---14.

7.CawsonRA,OdellEW,PorterSR.Neoplasticandnon-neoplastic diseasesofsalivaryglands.In:Cawson’sessentialsoforal pathol-ogyandoralmedicine.7thed.Edinburgh:ChurchillLivingstone; 2002.p.291---3.

8.JensenJL.Idiopathicdiseases.In:EllisGL,AuclairPL,GneppDR, editors.Surgicalpathologyofthesalivaryglands.Philadelphia: W.B.Saunders;1991.p.60---82.

Imagem

Figure 1 (A) Computed tomography scan (axial aspect) revealing a mineralized tissue with heterogeneous density and dimensions of 3.0 × 1.0 cm, approximately
Figure 4 The catheter and the sutures are in the correct surgical placement.

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